TY - JOUR
T1 - A case of Adrenocoricotrophic hormone -independent bilateral adrenocortical macronodular hyperplasia concomitant with primary aldosteronism
AU - Tokumoto, Mao
AU - Onoda, Naoyoshi
AU - Tauchi, Yukie
AU - Kashiwagi, Shinichiro
AU - Noda, Satoru
AU - Toi, Norikazu
AU - Kurajoh, Masahumi
AU - Ohsawa, Masahiko
AU - Yamazaki, Yuto
AU - Sasano, Hironobu
AU - Hirakawa, Kosei
AU - Ohira, Masaichi
N1 - Publisher Copyright:
© 2017 The Author(s).
PY - 2017/9/6
Y1 - 2017/9/6
N2 - Background: Adrenocoricotrophic hormone (ACTH) - independent bilateral adrenocortical macronodular hyperplasia (AIMAH) is a rare cause of Cushing's syndrome, and is characterized by bilateral adrenal hyperplasia. However, Primary aldosteronism (PA) is a relatively common adrenal disease. Case presentation: A 56-year-old man who has been treated hypertension and diabetes mellitus was detected low plasma potassium level with an elevated level of plasma aldosterone concentration and bilateral adrenal swelling. Endocrinological examinations showed autonomous secretion of cortisol and aldosterone, with suppression of plasma ACTH level and renin activity. A selective adrenal venous sampling demonstrated that left adrenal gland was responsible for aldosterone hypersecretion. He was diagnosed preclinical Cushing's syndrome due to ACTH - independent bilateral adrenocortical macronodular hyperplasia (AIMAH) associated with aldosterone producing adenoma of the left adrenal gland. A laparoscopic left adrenalectomy was performed. Conclusion: The resected adrenal specimen histologically consisted with a diagnosis of AIMAH. Moreover, tiny cell clusters positive immunostaining for aldosterone synthase was revealed. This is a rare case of AIMAH accompanied by preclinical Cushing's syndrome and primary aldosteronism.
AB - Background: Adrenocoricotrophic hormone (ACTH) - independent bilateral adrenocortical macronodular hyperplasia (AIMAH) is a rare cause of Cushing's syndrome, and is characterized by bilateral adrenal hyperplasia. However, Primary aldosteronism (PA) is a relatively common adrenal disease. Case presentation: A 56-year-old man who has been treated hypertension and diabetes mellitus was detected low plasma potassium level with an elevated level of plasma aldosterone concentration and bilateral adrenal swelling. Endocrinological examinations showed autonomous secretion of cortisol and aldosterone, with suppression of plasma ACTH level and renin activity. A selective adrenal venous sampling demonstrated that left adrenal gland was responsible for aldosterone hypersecretion. He was diagnosed preclinical Cushing's syndrome due to ACTH - independent bilateral adrenocortical macronodular hyperplasia (AIMAH) associated with aldosterone producing adenoma of the left adrenal gland. A laparoscopic left adrenalectomy was performed. Conclusion: The resected adrenal specimen histologically consisted with a diagnosis of AIMAH. Moreover, tiny cell clusters positive immunostaining for aldosterone synthase was revealed. This is a rare case of AIMAH accompanied by preclinical Cushing's syndrome and primary aldosteronism.
KW - ACTH - Independent bilateral adrenocortical macronodular hyperplasia
KW - Laparoscopic unilateral adrenolectomy
KW - Preclinical Cushing's syndrome
KW - Primary aldosteronism
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U2 - 10.1186/s12893-017-0293-z
DO - 10.1186/s12893-017-0293-z
M3 - Article
C2 - 28877721
AN - SCOPUS:85028911789
VL - 17
JO - BMC Surgery
JF - BMC Surgery
SN - 1471-2482
IS - 1
M1 - 97
ER -